Human pegivirus‐1 associated leukoencephalitis: Clinical and molecular features

Etiologic diagnosis is uncertain in 35% to 50% of patients with encephalitis, despite its substantial global prevalence and disease burden. We report on 2 adult female patients with fatal leukoencephalitis associated with human pegivirus‐1 (HPgV‐1) brain infection. Neuroimaging showed inflammatory c...

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Bibliographic Details
Published in:Annals of neurology 2018-11, Vol.84 (5), p.781-787
Main Authors: Balcom, Erin F., Doan, Matthew A.L., Branton, William G., Jovel, Juan, Blevins, Gregg, Edguer, Beste, Hobman, Tom C., Yacyshyn, Elaine, Emery, Derek, Box, Adrian, van Landeghem, Frank K.H., Power, Christopher
Format: Article
Language:English
Subjects:
Astrocytes
Brain
Diagnosis
Differential diagnosis
Encephalitis
Encephalitis - pathology
Encephalitis - virology
Etiology
Fatal Outcome
Female
Flavivirus
Flavivirus Infections - pathology
Gene deletion
Gene sequencing
Gliosis
Humans
Infiltration
Inflammation
Leukoencephalitis
Leukoencephalopathies - pathology
Leukoencephalopathies - virology
Lymphocytes
Medical imaging
Middle Aged
Neuroimaging
Neurology
Oligodendrocytes
Patients
Phylogeny
Ribonucleic acid
RNA
Substantia alba
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Summary:Etiologic diagnosis is uncertain in 35% to 50% of patients with encephalitis, despite its substantial global prevalence and disease burden. We report on 2 adult female patients with fatal leukoencephalitis associated with human pegivirus‐1 (HPgV‐1) brain infection. Neuroimaging showed inflammatory changes in cerebral white matter. Brain‐derived HPgV‐1 RNA sequences clustered phylogenetically with other pegiviruses despite an 87‐nucleotide deletion in the viral nonstructural (NS)2 gene. Neuropathology disclosed lymphocyte infiltration and gliosis predominantly in brain white matter. HPgV‐1 NS5A antigen was detected in lymphocytes as well as in astrocytes and oligodendrocytes. HPgV‐1 neuroadaptation should be considered in the differential diagnosis of progressive leukoencephalitis in humans. Ann Neurol 2018;84:789–795
ISSN:0364-5134
1531-8249
DOI:10.1002/ana.25343